What interests young autistic children? An exploratory ...

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RESEARCH ARTICLE

What interests young autistic children? An exploratory study of object exploration and repetitive behavior

Claudine JacquesID1,2*, Vale? rie Courchesne2, Andre? e-Anne S. Meilleur2, Suzanne Mineau2, Ste? phanie Ferguson2, Dominique Cousineau3, Aure? lie Labbe4, Michelle Dawson2, Laurent Mottron2,5

1 Psychoeducation and Psychology Department, Universite? du Que? bec en Outaouais, Gatineau, Que? bec, Canada, 2 Autism Research Group, CIUSSS du Nord-de-l'^ile-de-Montre?al, Montre? al, Que? bec, Canada, 3 Development Center, Centre Hospitalier Universitaire Ste-Justine, Montre?al, Que?bec, Canada, 4 HEC Montre?al, Montre? al, Que? bec, Canada, 5 Psychiatry Department, Universite? de Montre?al, Montre? al, Que? bec, Canada

* claudine.jacques@uqo.ca

Abstract

OPEN ACCESS

Citation: Jacques C, Courchesne V, Meilleur A-AS, Mineau S, Ferguson S, Cousineau D, et al. (2018) What interests young autistic children? An exploratory study of object exploration and repetitive behavior. PLoS ONE 13(12): e0209251.

Editor: Marina A. Pavlova, University Hospital of Tu?bingen, GERMANY

Received: February 19, 2018

Accepted: December 3, 2018

Published: December 31, 2018

Copyright: ? 2018 Jacques et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Behaviors characterized as restricted and repetitive (RRBs) in autism manifest in diverse ways, from motor mannerisms to intense interests, and are diagnostically defined as interfering with functioning. A variety of early autism interventions target RRBs as preoccupying young autistic children to the detriment of exploration and learning opportunities. In an exploratory study, we developed a novel stimulating play situation including objects of potential interest to autistic children, then investigated repetitive behaviors and object explorations in 49 autistic and 43 age-matched typical young children (20?69 months). Autistic children displayed significantly increased overall frequency and duration of repetitive behaviors, as well as increased specific repetitive behaviors. However, groups did not significantly differ in frequency and duration of overall object explorations, in number of different objects explored, or in explorations of specific objects. Exploratory analyses found similar or greater exploration of literacy-related objects in autistic compared to typical children. Correlations between repetitive behaviors and object explorations (their frequency and duration) revealed positive, not negative, associations in both groups. Our findings, from a novel situation incorporating potential autistic interests, suggest that RRBs do not necessarily displace exploration and its possibilities for learning in autism.

Data Availability Statement: Data cannot be shared publicly because the restrictions of Riviere des Prairies Hospital's research ethic committee prevent the open sharing of the full de-identified data set, even when participants are referred to by a code and their identity is concealed. Only the clinician-researchers (CJ, LM, SM and VC), the research assistants, and the two ethical committees can have access to the entire individual data, and the Research Ethics Board has determined there are restrictions on sharing deidentified data, as well. For more information about

Introduction

Behaviors, activities, or interests considered to be "restricted" and "repetitive" (RRBs) are one of two core domains which constitute the diagnosis of autism [1]. RRBs diagnostic of autism are diverse, encompassing motor mannerisms, repetitive use of speech or objects, insistence on sameness, atypical sensory responses, and intense interests, among others. In DSM-5 autism criteria, RRBs range from behaviors causing "significant interference with functioning

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restrictions on the dataset, contact Comite? D'E?thique Recherche Hrdp (CIUSSS NIM): Comite. Ethique.Recherche.Hrdp@ssss.gouv.qc.ca.

Funding: This study was funded by Canadian Institutes of Health Research grant MOP-130277 and by the Chaire de Recherche Marcel et Rolande Gosselin.

Competing interests: The authors have declared that no competing interests exist.

in one or more contexts" (Level 1) to those which "markedly interfere with functioning in all spheres" (Level 3 [2]). In much of the literature, RRBs are viewed as detrimental to the progress and functioning of autistic individuals, as well as distressing to their families (e.g.[3?5]). For instance, an "optimal outcome" in autism, as currently defined, is in part achieved through loss of all but minimal, i.e. typical level, RRBs [6].

However, studies investigating associations between RRBs and various outcomes in autism have had mixed results [7, 8], including inconsistent associations between RRBs and measured intelligence depending on how RRBs are assessed [9]; no association between RRBs and adaptive behavior in "minimally verbal" children [10]; no association between RRBs and socialcommunication scores in preschool children [11]; and a positive association between intense ("circumscribed") interests and non-verbal IQ (see also[12, 13]). RRBs did not predict language outcomes in speech-delayed autistics [14], and in early intervention studies, increased RRBs over time have been compatible with increased measured social abilities [15, 16] and increased measured intelligence [17]. The full range of evidence suggests that associations between RRBs and functioning in autism may be complex and not straightforwardly negative.

A particular concern has been that RRBs reduce autistics' attention to and exploration of their environment, depriving them of meaningful input and opportunities for learning starting early in life [8]. Restricted object use, where exploration is confined to a small number and limited variety of objects, is considered to limit their learning opportunities [18, 19]. Sensory hyper- or hypo-reactivity and unusual sensory interests, which feature in DSM-5 RRB criteria, can all be seen as limiting exploration in autism [20?22], as can motor deficits [23]. Nevertheless, direct evidence of reduced exploration in autism, and its relationship with RRBs, remains sparse. Pierce and Courchesne [24] compared the object explorations of small groups of preschool autistic and age-matched typical children. Exploration was assessed in a room with 10 kinds of boxes and bags (e.g. "large shopping bag with handles wrapped in multicolored wrapping paper"), 6 of which contained an array of arbitrary objects (e.g. "a plastic sack filled with blue water and a plastic fish"). In this situation, where their potential interests were not considered, preschool autistic children showed decreased object exploration negatively correlated at a trend level with their repetitive behaviors. Using the same paradigm but with larger agematched groups of toddlers, Bacon, Courchesne [25] again found fewer object explorations in autistics, as well as an altered quality of exploration rated as less appropriate and more stereotypical (e.g. "spinning wheels or visual inspection of objects").

Among RRBs in autism, atypical or intense ("circumscribed") interests may be the most commonly observed [13] yet have been the least studied across development. Evidence on relations among autistic interests, exploration, learning opportunities, and functioning remains sparse and disparate. In questionaires or interviews, the intense interests of autistic children were reported by their parents to cause functional impairment and great interference with activities [26, 27]; the intense interests of typical young children were, in contrast, positively perceived with no mention of interference even when such interests dominate children's lives [28]. In eye-tracking studies using images of parent-reported general categories of autistic interests (e.g. vehicles), preschool autistic children showed an interest-driven pattern of visual exploration interpreted as circumscribed and preserverative [29, 30]. A similar study in older children and adolescents found that autistics visually explored fewer objects with longer fixations, but increased object exploration was correlated with increased RRBs [31]. In adolescents and adults performing a selective attention task, there is also preliminary evidence that in some circumstances autistics may experience less distraction or interference from their intense interests than do typical individuals [32]. This result was contrary to predictions and suggests the importance of improved and more direct tests of when or whether RRBs, which encompass

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atypical autistic interests, limit or interfere with exploration and attendant learning opportunities in autism.

In this exploratory study, we developed a novel stimulating play situation incorporating objects of potential interest to young autistic children. It was used to investigate whether object exploration in this population is reduced in duration, frequency, or variety and/or complexity of objects explored. We assessed the frequency and duration of overall repetitive behaviors and object explorations and analyzed how they were associated, without excluding the possibility that exploring objects is one thing autistics may do during repetitive behaviors. We also assessed specific repetitive behaviors, as well as how many different objects were explored and which ones. Using different levels of structure in the play situation, we investigated whether exploration occurred spontaneously or required structured support. Young autistic children, regardless of their developmental test scores, were compared to age-matched typical children.

Method

Participants

The full sample included 92 children, 49 autistic and 43 typical, aged from 20 to 69 months. Autistic children were recruited from the autism database of Riviere des Prairies Hospital (HRDP) in Montreal. Autism diagnosis was established by a multidisciplinary team of expert clinicians using a comprehensive assessment, DSM-IV-TR or DSM-5 autism criteria, and clinical best-estimate judgment; all autistic participants scored above Autism Diagnostic Observation Schedule (generic or second edition version) autism spectrum cut-offs. None of the autistic children had another primary DSM-based diagnosis or an identifiable genetic condition. Typical children were recruited from a local daycare center. A questionnaire completed by parents confirmed that children in the typical group did not exhibit suspected or diagnosed autism, developmental delays, or behavioral issues (questionnaires missing for 4/43 typical children). While autistic and typical children were matched on age and sex, their performance on Mullen Scales of Early Learning-MSEL (data available for 40 autistic and 40 typical children, [33]) differed, as expected (e.g.[34, 35]), with fine motor, visual reception, receptive language, expressive language, and composite scores significantly lower in the autistic group (all p's ................
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